Alice Huault, Gérard Michel, Valérie Charon, Kamal Chouklati, Carine Domenech, Pascal Chastagner, Jean-Hugues Dalle, Catherine Paillard, Stéphane Ducassou, Marilyne Poirée, Geneviève Plat, Marie-Dominique Tabone, Justyna Kanold, André Baruchel, Claire Berger, Isabelle Pellier, Dominique Plantaz, Alexandre Theron, Alaa Mustafa, Pascal Auquier, Virginie Gandemer
Pediatr Hematol Oncol. 2023;40(5):458-474
https://pubmed.ncbi.nlm.nih.gov/36820621/
Abstract
Osteonecrosis (ON) is a known complication of acute leukemia (AL) management, affecting 1%-10% of young patients and resulting in long-term morbidity. Widespread access to MRI over the past decade has allowed earlier detection and more accurate assessment. This study investigated clinical and MRI features of the 129 (2.5%) patients with symptomatic ON retrospectively recruited from the French LEA (Leucémies de l’Enfant et de l’Adolescent, or child and adolescent leukemias) cohort (n = 4,973). We analyzed data concerning ON risk factors, multifocal involvement, severe lesions detected by MRI, and patient quality of life (QoL). ON patients tended to be >10 years old at the time of AL diagnosis (odds ratio [OR]: 22.46; p < 10-6), female (OR: 1.8; p = 0.002), or treated for relapse (OR: 1.81; p = 0.041). They more frequently suffered from other sequelae (p < 10-6). Most necroses involved weight-bearing joints, and they were multifocal in 69% of cases. Double-blinded review of MRIs for 39 patients identified severe lesions in 14, usually in the hips. QoL of adolescents and adults was poor and permanently impacted after onset of ON. In conclusion, age >10 at time of AL diagnosis, female sex, and relapse occurrence were risk factors for multifocal ON; MRI revealed severe ON in a third of the patients considered; and ON was associated with persistently poor QoL affecting multiple domains. Future studies should include prospective data addressing ON management and seek to identify genetic markers for targeted screening enabling early ON detection and treatment.
Keywords: Children; late effects; leukemia; osteonecrosis.